Seroprevalence of HTLV-I and HTLV-II among Intravenous Drug Users and Persons in Clinics for Sexually Transmitted Diseases

Abstract

Introduction Langerhans cell histiocytosis (LCH) is a clonal neoplastic proliferation of Langerhans cells with unclear etiology. LCH is a rare entity, with 1 per 1,5 million. Mostly it occurs in children and adolescents but in some cases a late onset could be observed. Only 6% of the cases affect the spine, and only one literature report has published its multiplex spinal appearance so far. The multifocal, progressive disease would require early histological diagnosis and oncological treatment to avoid its spreading and systemic, severe consequences. Materials and methods: Case report with four-year followup. Results: A young female patient (35y) with no past medical history presented with intermittent costal, back and low back pain. Detailed imaging studies described multiplex, diverse bone lesions in ThVIII, LIV, LV and iliac crest. MRI T2 signal intensity was varied from hypo-to hyperintensity. LIV showed a mixed lesion with contrast enhancement and soft tissue mass in the spinal canal without neurological compression. PET/CT proved a highly increased FDG uptake in more spinal sites (ThII, ThIV, ThVIII, ThIX, LI, LIV, LV) and the iliac crest. Infectious origin was excluded by detailed laboratory checkup. An LIV open biopsy was performed two months after the date of the MRI. Result was negative. Three month later a trocar biopsy was done from the radiologically significant iliac crest lesion but it did not result in any diagnosis. Laboratory tests, endocrinological and hematological detailed examinations showed no metabolic, hormonal or hematological disease. Repeated imaging studies showed a rapid change in the morphology of the bone lesions, but the patient complaints reduced and she refused any further invasive procedures. Two years later, the patient was readmitted into the hospital because of an acute, severe local neck pain without neurological signs. A severe, unstable CVI compression fracture was diagnosed and a CVI corpectomy was performed. The histological diagnosis of the fractured vertebral bone proved Langerhans cell histiocytosis. Oncological treatment (citarabin) was started 4 weeks after the surgery. One year after the surgery, local control and function is excellent, but complete remission has not been reached regarding the activity of the multiplex spinal lesions. Conclusion: Multiplex spinal LCH in adulthood is an extreme rare clinical entity and can cause severe pathological vertebral fractures. Our case report highlights the diagnostic difficulties of this tumor-like lesion. Based on the long-term follow-up of this patient, we can conclude that a very short interval is required between the imaging studies and the biopsy based on them to catch the bone lesion in its histologically positive phase and to start the early, effective oncological treatment.